Infliximab treatment for refractory COVID-19-associated multisystem inflammatory syndrome in a Japanese child. Issue 6 (June 2022)

• Record Type: Journal Article
• Authors: Yamaguchi, Yohei; Takasawa, Kei; Irabu, Hitoshi; Hiratoko, Kanako; Ichigi, Yosuke; Hirata, Ko; Tamura, Yumie; Murakoshi, Miki; Yamashita, Motoi; Nakatani, Hisae; Shimoda, Masuhiro; Ishii, Taku; Udagawa, Tomohiro; Shimizu, Masaki; Kanegane, Hirokazu; Morio, Tomohiro
• Is Part Of: Journal of infection and chemotherapy. Volume 28:Issue 6(2022)
• Page Start: 814
• Page End: 818
• Publication Date: 2022-06
• Subjects: Multisystem inflammatory syndrome in children -- Infliximab -- COVID-19 -- Hyperinflammation; Chemotherapy -- Periodicals; Infection -- Periodicals; Communicable diseases -- Chemotherapy -- Periodicals; 615.5805
• DOI: 10.1016/j.jiac.2022.01.011
• Languages: English
• ISSNs: 1341-321X

Abstract

Abstract: Patients with multisystem inflammatory syndrome in children (MIS-C) can develop clinical features resembling Kawasaki disease (KD). A full picture of MIS-C in East Asia which has higher incidence of KD than other regions remains unclear. We report on a 15-year-old Japanese boy with refractory MIS-C who was successfully treated with infliximab. A Japanese boy who was diagnosed with coronavirus disease 2019 (COVID-19) before a month developed MIS-C with fulfilling six principal symptoms of KD. Laboratory data showed extreme hyperferritinemia (11, 404 ng/mL), besides lymphopenia and thrombocytopenia. The patient was refractory to initial therapy with intravenous immunoglobulin (IVIG; 2 g/kg), aspirin, and prednisolone. He was therefore administered a second IVIG (2 g/kg) and infliximab (5 mg/kg) on days 7 and 8 from the onset of fever, respectively, which resulted in an improvement of clinical symptoms. Only four Japanese cases with MIS-C were reported and all of them were responsive to IVIG. The hyperferritinemia in this case was distinctive from previously reported MIS-C cases in Japan and other cohorts and may be associated with refractoriness to IVIG therapy. Marked elevation of circulating ferritin levels is known to be induced by tumor necrosis factor-α, which plays a key role in the pathogenesis of both KD and MIS-C. Thus, for MIS-C patients with hyperferritinemia, early intervention with adjunctive infliximab may induce a more rapid resolution of inflammation and improve outcome. Because MIS-C may be heterogeneous with respect to immunopathology, genetic background, clinical phenotypes and response to therapies, optimized treatment strategies according to immunopathogenesis are required.